This lesion can involve either jaws, but the anterior region of the mandible is the most affected area

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口腔病理科 On-Line KMU Student Bulletin

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原文題目(出處)： Glandular odontogenic cyst: a case report. 2008;1:223-7 原文作者姓名： de Castro JFL, Henriques ACG, Medeiros MD, Montenegro

LT, Cazal C

通訊作者學校： Oral Pathology Department, School of Dentistry, Federal University of Pernambuco, Pernambuco, Brazil

報告者姓名(組別)：鄧宇廷 Intern L組

報告日期： 2009/9/8

內文：

 Abstract

◎ GOC is a rare developmental lesion considered a distinct entity because of its uncommon histopathological characteristics.

◎ This lesion can involve either jaws, but the anterior region of the mandible is the most affected area.

◎ It strikes distinct age groups, with an average patient age of 50 years.

◎ The aim of the present article is to report a case of GOC in a 40-year-old female patient exhibiting an intrabone cyst in the anterior mandibular region.

 Introduction

◎ The term glandular odontogenic cyst (GOC) has been proposed as the most appropriate designation for this rare pathological lesion.( It was formerly denominated mucous-producing odontogenic cyst and sialoodontogenic cyst because of a probable origin from the tissue which forms the salivary glands.)

◎ The main published features include the presence of a radiolucent swelling in the jaws, a tendency towards local recurrence after conservative surgical treatment.

◎ Radiography reveals a unilocular or multilocular radiolucent image with well-defined margins. Other radiographic characteristicswhich can be presented in GOC include: root displacement and resorption of the teeth involved as well as tapering, erosion and perforation of the cortical bone.

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◎ The epithelial cells may form circular proliferations resembling spheres, or spherical nodules, which are a significant characteristic, although not always present.

◎ Other characteristics include microcysts or intraepithelial crypts containing mucin, mucous cells and hyaline bodies, within the thickest section of the epithelium and in the lumen of the cyst.

◎ An uncommon presence of calcifications, phantom cells, foreign multinuclear giant cells and a chronic inflammatory infiltrate in the wall of the cyst might be also observed.

◎ Binda et al.3 and Ferreira Jr et al.10 observed an interesting histological characteristic: epithelial proliferation within the cyst wall, which may suggest a neoplasic potential and a possible association with central mucoepidermoid carcinoma (MEPCa).

◎ The GOC was located in the anterior region of the mandible, mimicking a lesion of an endodontic origin or a pseudocyst. The diagnosis of GOC was confirmed through histopathological examination.

 Case Report

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During a routine radiographic exam, a 40-year-old female patient presented an asymptomatic intrabone

lesion. It was located in the anterior area of the mandible near the periapical region of the 41, 31 and 32 tooth. The radiography revealed a unilocular radiolucent image with well-defined margins and radiopaque sclerotic edges (Fig. 1). The initial diagnostic hypothesis was of traumatic bone cyst. Because it was not a

periapical inflammatory lesion of endondontic origin, patient was informed to proceed with surgical removal of the cyst. However, the patient did not return to give continuity to the suggested treatment. Upon returning 4 months after the initial diagnosis, the patient returned with the 41 teeth endodontically treated. The oral surgeon was able to give continuity to the treatment and to perform the complete surgical removal of the lesion with ample curettage (Fig. 2). The specimen was formalin fixed and sent to the oral pathology laboratory where the histopathological analysis was performed. The anatomopathological examination revealed fragments of a cystic lesion of odontogenic nature compatible with GOC. The epithelial lining exhibited cuboidal or columnar cells with a stratified aspect in which areas of papillary irregularity could be observed (Fig. 3). Duct-like microcystic spaces were observed bordered by cuboidal cells (Fig. 4). Mucous cells were also seen. The cyst sac was composed of richly cellularised and vascularised fibrous conjunctive tissue. The PAS staining revealed positive mucous cells and occasional cystic

spaces containing positive PAS material (Fig. 5). Figure 5 Photomicrography exhibiting PAS-positive eosinophilic material

in the interior of the microcystic structures (arrow). Some PAS-positive mucous cells are also present (asterisk).

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The AE1/AE3 antibody was positive (Fig.

6), and the proteins p53 and Ki-67 were strongly expressed throughout the length of the epithelia, respectively confirming the deregulation of the tumour suppression mechanism (Fig. 7) and the presence of mitotic activity (Fig. 8). The patient’s follow up has been carried out for 18 months and she has not presented any recurrences thus far. Figure 9 shows the initial bone regeneration after 1 year from the surgical treatment.

 Discussion

The importance of knowledge regarding the intrabone lesions that share the differential diagnosis with GOC.

The radiographic characteristics observed in GOC can lead to a diagnosis of keratocystic odontogenic tumour, ameloblastoma, central lesion of giant cells, lateral periodontal cyst, root cyst, simple bone cyst, aneurysmatic bone cyst, periapical cementum dysplasia, ossifying fibroma, odontogenic myxoma and central low-grade MEPCa.

題號題目

1 腺體齒源性囊腫(Glandular odontogenic cyst)的病理組織發現可能與

下列何種唾液腺腫瘤相似?

(A) 造釉細胞瘤(ameloblastoma)

(B) 鈣化上皮齒源瘤(calcifying epithelial odontogenic tumor) (C) 黏液上皮樣癌(mucoepidermoid carcinoma)

(D) 多行性腺瘤(pleomorphic adenoma)

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答案(C) 出處：Oral & Maxillofacial Pathology, P.607

題號題目

2 下列何囊腫歸於發育性囊腫?

1. 含齒囊腫(Dentigerous cyst)

2. 齒源性角化囊腫(odontogenic keratocyst) 3. 戈林囊腫(Gorlin cyst)

4. 腺體齒源性囊腫(Glandular odontogenic tumor)

(A) 1+2

(B) 3+4

(C) 1+3+4

(D) 1+2+3+4

答案(D) 出處：Oral & Maxillofacial Pathology, P.494